key: cord-0791664-inqu6nan authors: Aihole, Jayalaxmi Shripati title: Early neonatal lobectomy for ILE; is it feasible?() date: 2021-08-26 journal: Int J Surg Case Rep DOI: 10.1016/j.ijscr.2021.106351 sha: 6aaa0bf293afd758bf5bab30954dee766022ad8b doc_id: 791664 cord_uid: inqu6nan INTRODUCTION: Congenital lobar emphysema (CLE) also called congenital lobar overinflation and infantile lobar emphysema is a rare congenital anomaly of lung often presents in the neonatal period, with hyperinflation of one or more pulmonary lobes. IMPORTANCE: Author is reporting here an uneventfully done earliest ever neonatal lobectomy for ILE. Nelson RL in 1932 first described congenital lobar emphysema (CLE). Its prevalence is 1 in 20,000 to 1 in 30,000. Though its aetiology is not well understood yet, CLE is a rare condition causing over inflation of lung lobes, compressing the ipsilateral, as well as the contra lateral lung; causing acute respiratory distress. Often detected in neonates or young infants but, some even present late at childhood. The upper lobes are affected in over 95% of cases, with multiple lobes in about 15% of cases. A day 1 male neonate, born full term by lower segment caesarean section with a birth weight of 2.68 kg was noticed to have respiratory distress immediately after birth. Chest radiography revealed hyperlucent left lung with mediastinal shift; contrast enhanced computed tomography (CECT) showed left hyperinflated lung with gross mediastinal shift (Fig. 1A , B, C). Baby was clinically stable requiring oxygen of 2 l per minute without ventilator support. Having normal haematological parameters as well as reverse transcriptase polymerase chain reaction (RT PCR) test negative for severe acute respiratory syndrome corona virus 2 (SARS-CoV2), baby was taken up for left lobectomy under general anaesthesia. Induction done with thiopenone, fentanyl, propofol, atracurim and intubed with 3 mm micro cuffed endotracheal tube; maintained with oxygen, air, isoflurane and intermittent positive pressure ventilation (IPPV) with bilateral lungs kept ventilated throughout the procedure. Left posterolateral thoracotomy and left upper lobe ILE was exposed, its supplying vessels and bronchus were ligated at the hilar level, cut and resected ILE sent for histopathology, which revealed ILE showing massive distension of alveoli without tissue destruction (Fig. 2E ,F,G,H, I). Post operatively baby improved uneventfully without any ventilator support and hence discharged on post operative day 5. Infantile lobar emphysema (ILE) occurs due to over distension of a pulmonary lobe, result of a partial postnatal obstruction of the associated bronchus with air trapping. Hence baby presents with respiratory distress, 50% in the first week and in 80% first 6 months of life [1, 2] . Imaging studies usually show hyperlucent lobe or a lobe of normal lucency that occupies a disproportionate part of the hemithorax, with mediastinal shift and compression of the uninvolved lobes. Associated anomalies are present in 5% to 40% of patients, with 70% of these anomalies are cardiovascular. The supplying bronchus may reveal stenosis, atresia, or intrinsic obstruction, although many cases may be caused by extrinsic bronchial obstruction, so the bronchus may appear grossly normal [1] [2] [3] . Microscopically the lung shows a uniform over distension of apparently normally developed acini with alveolar saccules and alveoli 3 to 10 times the normal size (Fig. 2H, I) [1, 3, 4] . Congenital lobar emphysema (CLE) may be clinically confused with tension pneumothorax. Hence the chest tube insertion may further worsen the distress and lead to further lung injury. In CLE the pulmonary vessels extend to the periphery of the hyper inflated lobe and there is no visualization of a pleural line compared to pneumothorax [2, 3] . The differential diagnosis includes congenital cystic adenomatoid malformation, sequestration, bronchogenic cyst, unilateral hyper lucent lung syndrome, and pulmonary interstitial emphysema. Though conservative management has supported by some studies due to its potentially reversible status in few cases which is very rare event but difficult to predict, hence surgery is the most definitive and curative; either via segmentectomy or lobectomy [1] [2] [3] [4] [5] . Nevertheless symptomatic respiratory distress in a neonate needs definitive surgical management to avoid morbidity and mortality. Though the literature is abundant with ILE with varied time for its definitive resection of the affected lobe, either by open or by minimally invasive approach, author is reporting the first ever, lobectomy done for a case of ILE on a post natal day 5 uneventfully; in a tertiary care neonatal and paediatric centre. Having adequate facilities with normal haematological parameters, early neonatal lobectomy is feasible option in a tertiary care centre. Not commissioned, externally peer-reviewed. None. Yes taken. Written informed consent was obtained from the parents for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request. Not yet done. Dr Jayalaxmi Shripati Aihole. Nil. Dr Jayalaxmi Shripati Aihole has contributed sole to the artilce. Congenital lobar emphysema: 30-year case series in two university hospitals Congenital lobar emphysema: a diagnostic challenge and cause of progressive respiratory distress in a 2 month-old infant Surgical management of infants with congenital lobar emphysema and concomitant congenital heart disease Congenital lobar emphysema: differential diagnosis and therapeutic approach The SCARE 2020 guideline: updating consensus Surgical CAse REport (SCARE) guidelines Author would like to thank all her paediatric surgical colleagues, anesthetists, OT staffs, and radiology staffs of IGICH Bengaluru, Karnataka.Manuscript has not been previously published in whole or in part or submitted elsewhere for review. None.